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Adler et al.

BMC Pregnancy and Childbirth 2013, 13:246


http://www.biomedcentral.com/1471-2393/13/246

RESEARCH ARTICLE

Open Access

Estimating the prevalence of obstetric fistula:


a systematic review and meta-analysis
A J Adler*, C Ronsmans, C Calvert and V Filippi

Abstract
Background: Obstetric fistula is a severe condition which has devastating consequences for a womans life. The
estimation of the burden of fistula at the population level has been impaired by the rarity of diagnosis and the lack
of rigorous studies. This study was conducted to determine the prevalence and incidence of fistula in low and
middle income countries.
Methods: Six databases were searched, involving two separate searches: one on fistula specifically and one on
broader maternal and reproductive morbidities. Studies including estimates of incidence and prevalence of fistula at
the population level were included. We conducted meta-analyses of prevalence of fistula among women of
reproductive age and the incidence of fistula among recently pregnant women.
Results: Nineteen studies were included in this review. The pooled prevalence in population-based studies was
0.29 (95% CI 0.00, 1.07) fistula per 1000 women of reproductive age in all regions. Separated by region we found
1.57 (95% CI 1.16, 2.06) in sub Saharan Africa and South Asia, 1.60 (95% CI 1.16, 2.10) per 1000 women of reproductive
age in sub Saharan Africa and 1.20 (95% CI 0.10, 3.54) per 1000 in South Asia. The pooled incidence was 0.09 (95%
CI 0.01, 0.25) per 1000 recently pregnant women.
Conclusions: Our study is the most comprehensive study of the burden of fistula to date. Our findings suggest that
the prevalence of fistula is lower than previously reported. The low burden of fistula should not detract from their
public health importance, however, given the preventability of the condition, and the devastating consequences of
fistula.
Keywords: Vesicovaginal fistula, Maternal morbidity, Systematic review

Background
The World Health Organisation defines an obstetric fistula (referred to as fistula in the text below) as an abnormal opening between a womans vagina and bladder
and/or rectum through which her urine and/or faeces
continually leak [1]. Classifications of fistula vary, but
they generally include fistulae from obstetric causes including vesicovaginal fistula (VVF) and rectovaginal fistula (RVF). Fistulae have devastating consequences [2,3],
particularly in low income countries where women have
less geographical and financial access to appropriate surgical care for repair. In high income countries they are
also devastating, but they are very rare and surgery to
repair them occurs more rapidly.
* Correspondence: alma.adler@lshtm.ac.uk
London School of Hygiene & Tropical Medicine, Keppel St, London, WC1E
7HT, UK

In high income countries, fistulae are due to iatrogenic


causes; generally the result of radiation therapy and surgical interventions [4]. In low income countries where access to intrapartum care may be restricted, fistulae are
associated with a prolonged or obstructed labour, most
commonly occurring when a babys head becomes lodged
in the mothers pelvis cutting off blood flow to the surrounding tissues. Prolonged obstruction can cause the tissues to necrotise leading to fistula formation [2].
Women with fistulae often experience horrific or difficult associated conditions which stem either from the
fistulae itself or from the prolonged or obstructed labour
which caused it [3]. The most obvious consequences are
incontinence, either urinary [3], faecal or both. The constant leakage of urine and faeces can also lead to damage
to the vulva and thighs [2]. Fistulae are linked with social
ostracisation [5] and marginalisation [6]. Many case

2013 Adler et al.; licensee BioMed Central Ltd. This is an open access article distributed under the terms of the Creative
Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly cited.

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series show high rates of divorce or separation [7,8], absence of sexual intercourse [6,7], loss of fertility and
amenorrhea [9,10] and depression [8,11] among women
who have a fistula.
Fistulae are thought to have the highest prevalence
where maternal mortality is high, but there is great uncertainty about the actual prevalence [2]. In the 2000
Global Burden of Disease, Dolea and AbouZhar estimated that 0.08% of all births and 2.15% of neglected
obstructed labour births resulted in fistula [12]. These
estimates came from four studies only, all of which were
in sub-Saharan Africa and two were hospital-based. In
2006, the WHO estimated that more than 2 million
young women throughout the world live with untreated
fistula, and that between 50,000 and 100,000 new
women are affected each year [1]. These statistics originated from countries rapid needs assessments and physicians reports, mostly available in the grey literature,
and not from epidemiological studies using robust design, and almost none include a denominator.
In 2007, Stanton and colleagues wrote a paper [13] on
the challenges of quantifying fistula. They described
three types of publications reporting on frequency, incidence or prevalence of fistula. The first category of papers relied on secondary and tertiary citations (many of
which culminated in personal communications) and reported the number of patients treated without denominators. The second type of publications relied on

Cross-sectional and cohort studies with hospital based


recruitment

No
Exclude

Methods
A two-stage systematic review was conducted in accordance with the STROBE guidelines (http://www.strobestatement.org/) using free-text and subject headings in
Pubmed, Embase, Popline, Lilacs, WHOs Eastern Mediterranean database, and African Index Medicus published
until the end of 2012. The first stage targeted studies specifically reporting on fistula in the title, abstract or subject
headings, including search terms such as fistula, vesicovaginal fistula, and VVF. The second stage aimed at identifying additional studies that examined postpartum and
reproductive morbidity more broadly, whether or not fistula was specifically mentioned as a pathology. This search

Cross sectional and cohort community based studies

Clear
denominator and
adequate follow
up?

Yes
Include

declarations made by the authors themselves, or on surgeons estimates but the source of data was unclear.
The third type of studies described methods and provided appropriate denominators, but with varying degrees of transparency. Stanton and colleagues were only
able to find four papers in this third category [13].
The aim of our review is to provide improved estimates
of the prevalence and incidence of fistula by broadening
the search and including studies that may previously have
been overlooked. Unlike previous reviews of fistula
[12-14], we include studies that examine a broad spectrum
of reproductive morbidity (including morbidity studies
where fistula is but one of many outcomes studied) and/or
where no cases of fistula were reported.

Fistula studies

Reproductive
morbidity studies

Clear
denominator?

Include fistula
as morbidity?

Yes
Include

No
Exclude

Yes

Have Physician Exam


and include prolapse as
morbidity?

Have
Physician
Exam

Yes
Include

Figure 1 Types of studies included in our analysis.

No

No
Exclude

Yes
Include

No
Exclude

Adler et al. BMC Pregnancy and Childbirth 2013, 13:246


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included terms such as reproductive morbidity and maternal morbidity. A full search strategy is available upon request. Only English terms were used in the search, but
articles were not excluded based on language.
Reference lists were searched for additional articles.
Using Web of Science, all relevant articles were subjected
to forward citation searching to obtain further articles.
As illustrated in Figure 1, the following study designs
were included: (i) cross-sectional or cohort studies of fistula with hospital based recruitment of pregnant or recently delivered women where the women were followed
for and examined at least 30 days after the end of pregnancy and there was a clear denominator; (ii) cross sectional or cohort studies of prevalent or incident cases of
fistula in the community and (iii) cross sectional or cohort studies of prevalent or incident cases of reproductive morbidity in the community. Studies were only
included if women were examined for the presence of
fistula in hospital and/or if a well trained provider performed a physical examination of the genital area. Morbidity studies not reporting or mentioning fistulae but
including a robust design, a thorough physical exam of
the genital area that reported cases of uterine prolapse
were assumed to have zero cases of fistula, as it was assumed that if fistula had been found it would have been
reported. Studies relying on womens self-reports were
excluded because self-reports of reproductive morbidity
have been shown to be unreliable [13,15,16]. We excluded studies that were conducted before 1990 or published before 1991. We included studies irrespective of
sample size but studies without a denominator were excluded. If more than one paper provided results of the
same study population, data were first extracted from
the article with the greatest amount of information,
and supplementary data extracted from the other papers if required.
Data were extracted by a single author (AJA) using a
proforma and included information on region, study dates,
study population, duration of fistula, type of fistula, risk
factors, associated complications and denominators,
how fistula were ascertained, sampling technique, number of women and number of deliveries. There are no
good tools for looking at study quality in crosssectional studies, so study quality was assessed using a
modified Ottawa-Newcastle score (http://www.ohri.ca/
programs/clinical_epidemiology/oxford.asp).
The studies reported on two types of populations of
women: women of reproductive age and women with a recent pregnancy. In studies targeting women of reproductive
age we calculated the prevalence of fistula per 1000 women
of reproductive age. In studies where women were followed
after end of pregnancy, we calculated the incidence of fistula per 1000 recently pregnant women (assuming that
women were unlikely to have had fistula before getting

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pregnant). In all cases 95% confidence intervals were calculated using the binomial exact method.
Meta-analyses were conducted using the metaprop
command from the R 2.15.3 package meta using a
random-effects model [17]. Meta-analyses were conducted
summarising the prevalence of fistula among women of
reproductive age and the incidence of fistula in recently
pregnant women. All studies were stratified by continent
and by recruitment in hospital or in the community.

Results
The initial search of studies focussing on fistula found
9130 references after de-duplication, 367 of which were
retained after title and abstract screening. From the 367
articles, six were found to have information on either
prevalence or incidence of fistula [18-23]. A further 13

Initial search 11,366

After removing
duplicates 9130

Obtained for full text


367

Papers with information


on incidence and
prevalence 6

Found from
general maternal
and reproductive
morbidity studies
10

Found from
forward
searching and
reference lists 10

Final: 19 Studies

Figure 2 PRISMA diagram of studies.

Author

Study design

Assessment of fistula

Number of fistula

Number of
women/
pregnancies

Prevalence
(per 1000 WRA)

Muleta et al., 2008 [18]

Seven rural administrative


regions in Ethiopia

Cross-sectional survey of
obstetric fistula

Women reporting leakage of


urine, faeces or both examined
in the health facilities

44 (untreated)

19,153

1.62 (1.53, 2.64)

Walraven et al., 2001 [24]

Random sample of 20 rural


villages in Farafenni, The
Gambia

Census of all women aged 15-54


for reproductive morbidity

External, vaginal speculum and


bimanual pelvic examination by
female gynaecologist

1,038

0.95 (0.02, 5.26)

Kulkarni, 2007 [35]

Six PHC areas (urban and


rural) in Maharashtra, India

Cross sectional survey of


non-pregnant, ever married women
with proven fertility
for reproductive morbidity

Clinical examination but


unspecified what or by whom

1,167

0.86 (0.02, 4.8)

Bhatia et al., 1997 [19]

Villages (25% urban, 75% rural)


with at least 500 people in
Karnataka, India

Cross sectional study of all eligible


women under 35 with a child
under 5 for reproductive morbidity

External, vaginal speculum and


bimanual pelvic examination by
female gynaecologist

385

2.6 (0.07, 14.39)

Younis et al., 1993 [29]

Two rural villages in Giza,


Egypt

Cross sectional study of


reproductive morbidity in
ever-married, non pregnant
women.

Speculum and bimanual


examination by female
physicians [1]

509

0.0 (0.0 , 7.90)

Deeb et al., 2003 [27]

Nabi Sheet, Lebanon

Cross sectional study of


reproductive morbidity in ever
married, non-pregnant women

Thorough inspection of external


genitalia, with speculum
conducted by female
physicians [1]

506

0.0 (0.0, 7.3)

Al-Riyami et al., 2007 [28]

Oman, Mixed

National Health Survey 2000 aiming


to identify reproductive morbidity.
Multi-stage stratified probabilitysampling design of 1,968 households
with ever married, non-pregnant women

Pelvic examination by a trained


physician [1]

1,662

0.0 (0.0, 2.2)

Al-Qutob, 2001 [26]

Ain Al-Basha, Jordan.


Semi-urban

Random sample of Jordanian


women

Comprehensive physical and


pelvic examination conducted
by trained female physician, a
nurse/midwife and a laboratory
technician [1]

379

0.0 (0.0, 9.7)

Bulut et al., 1995 [25]

City of Istanbul, Turkey

Systematic sample of non-pregnant,


ever married parous women who
had ever used contraception

Physical examination by female


physician [1]

696

0.0 (0.0, 5.3)

Tehrani et al., 2011 [34]

Four provinces of Iran

Multi-stage stratified probabilitysampling design of non-pregnant


non menopausal women 18-45

Comprehensive gynaecological
examination of all married
women including a speculum
examination [1]

1117

0.0 (0.0. 3.3)

Community based studies

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Study area

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Table 1 Characteristics of studies reporting fistula prevalence included in the review

Studies with hospital based recruitment


Ijaiya and Aboyeji, 2004 [23]

Ilorin, Nigeria, urban

Hospital review of women with fistula repair

Repair

34

32,188

1.1 (0.7, 1.5)

Kalilani-Phiri et al., 2010 [21]

Nine districts (urban and


rural) in Malawi

Hospital record reviews from gynaecological,


prenatal, obstetric wards and operating
theatres as well as fistula repair services.
Only women originating from nine
districts included

Repair

111

425,865

0.26 (0.2, 0.3)

Mabeya, 2004 [36]

West Pokot, Kenya. Rural

Hospital record review supplemented by


surgeons notes. Cases of fistulae
presenting to the two rural hospitals that
are the main hospitals in the district

Repair

66

150,000

0.44 (0.34, 0.55)

[1] These studies were reproductive morbidity studies which did not state in the methods that they were investigating fistula, nor did they report any cases of fistula; however the type of examination used to identify
other reproductive morbidities was assessed to have been sufficient that should there have been any cases of fistula they would have been identified.

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Table 1 Characteristics of studies reporting fistula prevalence included in the review (Continued)

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Author

Study area

Study design

Assessment of fistula

Number of
fistula

Number of women/
pregnancies

Incidence (per 1000


pregnant women)

Vangeenderhuysen et al.,
2001 [22]

Eight centres (urban and


rural) in six countries in
West Africa

Prospective cohort study of all


pregnant women found by a
door to door census of households
followed up from antepartum to
two months postpartum.

Women reporting gynaecological


problems. Fistula assessed at last
contact 60 days after delivery

19,694

0.10 (0.01, 0.3)

Ferdous et al., 2012 [33]

Matlab, Bangladesh. Rural

Prospective cohort of all women


with obstetric complications, a
perinatal death or caesarean
section, and random sample of
women with uncomplicated births.

Physical examination at health


centre from six to nine weeks
postpartum

1,162

0 (0, 3.17)

Fronczak et al., 2005 [20]

Urban slums in Dhaka,


Bangladesh

Prospective community-based study


of women completing at least seven
months of pregnancy. Women excluded
if birth identified more than 21 days
postpartum. Sample selected using
multi-stage probability.

Physical exam conducted by


female physicians conducted
one-month postpartum

557

0 (0.0. 6.6)

Community based studies

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Table 2 Characteristics of studies reporting fistula incidence included in the review

Studies with hospital based recruitment


Filippi et al., 2007 [30]

Seven public urban and rural


hospitals in Burkina Faso

All women with severe obstetric


complications delivering in hospitals
and two controls per case. Interviews
conducted at 3, 6, and 12 months
after pregnancy.

Medical examinations

1,014

0.99 (0.03, 5.48)

Filippi et al., 2010 [31]

Cotonou, Porto Novo and


neighbouring communities
in south Benin

Prospective cohort study of women


with severe obstetric complications
and a sample of women with
uncomplicated childbirth.

Medical examination with


obstetricians

709

1.41 (0.04, 7.83)

Prual et al., 1998 [32]

Niamey city, Niger. Urban

All deliveries in six maternity wards,


and all complications referred to the
two referral maternity wards occurring
from 28th week antenatal to 42nd day
postpartum.

Medical examinations

4,081

0.49 (0.06, 1.77)

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Figure 3 Prevalence of fistula per 1000 women of reproductive age.

studies [24-34] were found from searching general maternal and reproductive morbidity studies (including
three from reference lists and forward citation searching
[34-36]) (Figure 2).
Of the 19 studies, 13 were community-based; seven of
which reported on fistula [18-20,22,24,33,35] and six
did not mention fistula anywhere in the paper. Ten
community-based studies reported the prevalence of fistula among women of reproductive age (Table 1) and
three reported the incidence among recently pregnant
women (Table 2). Six studies recruited women in hospital and followed them for 30 days or more after the
end of pregnancy (Table 2). Three studies [21,23,36], report the prevalence of fistula among women after having
fistula repairs. These studies were included because it
was possible to understand the population that these
women came from, and have a denominator (Table 1).
The other three studies reported the incidence of fistula
among women after giving birth or miscarrying in hospital. These studies recruited women with obstetric
complications as well as a sample of women without
complications [30-32].

Three community-based studies were from sub-Saharan


Africa [18,22,24], four were from South Asia [19,20,33,35],
two were from North Africa [28,29], three from the Middle East [26,27,34], and one from Turkey [25]. All six
studies using hospital-based recruitment were from subSaharan Africa [21,23,30-32,36].
The prevalence of fistula in community-based studies
ranged from 0 to 1.62 per 1000 women of reproductive
age with a median of 0 per 1000 (Figure 3). The pooled
prevalence of fistula in community-based studies was
0.29 (10 studies including 34,505 participants 95%
CI 0.00, 1.07) per 1000 women of reproductive age
(Figure 3). The pooled estimate for sub-Saharan Africa
and South Asia was 1.13 (4 studies with 29,680 participants 95% CI 0.72, 1.61) per 1000 women of reproductive age (Figure 4). By continent Sub-Saharan Africa had
an overall prevalence of 1.60 (two studies including
28,128 participants 95% CI 1.16, 2.10) per 1000 women
of reproductive age and South Asia had a prevalence of
1.20 (two studies with 1552 participants 95% CI 0.10,
3.63) per 1000 women of reproductive age (Figure 4).
Because all studies from the Middle-East and North

Figure 4 Prevalence of fistula per 1000 women of reproductive age stratified by region.

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Figure 5 Prevalence of fistula per 1000 women of reproductive age in studies with hospital-based recruitment.

Africa had zero events, it is impossible for the metaanalysis to provide a meaningful estimate due to the manner with which zero prevalence studies are dealt with
(http://cran.r-project.org/web/packages/meta/meta.pdf).
The prevalence of fistula in studies with hospital-based
recruitment ranged from 0.26 to 1.06 per 1000 women
of reproductive age, with a median of 0.44 per 1000
women of reproductive age. The pooled estimate of the
prevalence of fistula in hospital was 0.51 (three studies
with 608,053 participants 95% CI 0.25, 0.87) per 1000
women of reproductive age (Figure 5).
The pooled incidence of fistula in community-based
studies was 0.09 (three studies with 21,413 participants
0.01, 0.25) per 1000 recently pregnant women (Figure 6)
and in hospital-based studies 0.66 (three studies including 5804 participants 0.16, 1.48) per 1000 recently pregnant women (Figure 7).
The I [2] values varied substantially by stratum, ranging
from 0% in community-based (Figure 6) and hospitalbased (Figure 7) incidence studies to 94.8% in prevalence
studies with hospital based recruitment (Figure 5). Study
quality in the modified Ottawa-Newcastle score table is
shown in Table 3. All studies had a cross-sectional or cohort design.
Only two studies reported information on duration of
fistula: one found a median of eight years [18], and the

other a median of three years [21]. No community based


studies reported on the mode of delivery of the women,
or on the cause of fistula.

Discussion
Our comprehensive systematic review found that fewer
than 1 per 1000 women of reproductive age in low and
middle income countries suffer from fistula; this figure
rises to 1.57 per 1000 when only data from sub-Saharan
Africa and South Asia is used. The number of new cases
of fistula ranged from 0.09 per 1000 recently pregnant
women in community-based studies to 0.66 per 1000
pregnancies in hospital-based studies.
The WHO has suggested that over two million women,
mostly from sub-Saharan African and Asian countries,
have fistula [1,37]. Given an estimated population of 645
million women of reproductive age in sub-Saharan Africa
and South Asia in 2010 (http://esa.un.org/wpp/unpp/
p2k0data.asp), this would suggest that 3 per 1000 women
of reproductive age have a fistula, which is considerably
higher than our estimate for low and middle income
countries. There were too few studies in our review to arrive at robust estimates of the prevalence of fistula by continent, but even the highest estimates for sub-Saharan
Africa (1.62 per 1000 women of reproductive age in
Ethiopia) or South Asia (2.6 per 1000 in India) fall well

Figure 6 Incidence of fistula per 1000 pregnancies in community-based studies, stratified by region.

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Figure 7 Incidence of fistula per 1000 pregnant women in studies with hospital-based recruitment.

short of the WHO estimates. One study [22] estimated an


incidence of 1.239 per 1000 deliveries in rural regions and
used this to approximate 33,451 new fistula a year in Sub
Saharan Africa. This was based on 2 fistulae found in the
rural region in the MOMA study. Elsewhere they refer
to these regions as small towns [38]. They then used the
sample of 2 fistulae per 1543 live births to estimate the
overall incidence for sub-Saharan Africa. This approach is
likely to have over-estimated the number of fistulae
patients. First because a substantial proportion of African
women now live in cities where the risk of fistulae is
likely to be lower than in smaller towns or rural areas;
second because the denominator does not include the
urban women in larger cities for whom there were no
fistulae occurrence.
Overall, we estimate that just over one million women
may have a fistula in sub-Saharan Africa and South Asia,
and that there are over 6000 new cases per year in these
two world regions. Given the devastating consequences
of fistula for women and their families, this represents a
very substantial burden.
It is possible that community based studies represent
an underestimate of the prevalence of fistula, as fistulae
are generally more commonly found in regions where
there is no access to obstetric care, and may be difficult
to reach. These women may have been missed in the
studies included here, and the estimates provided here
may represent a lower bound estimate of prevalence.
However both the studies in Ethiopia [18] and the
Gambia [24] were conducted in rural areas, and both
studies showed very low estimates of prevalence (1.62
and 0.96 respectively).
Our search identified 13 additional studies that would
not have been found had the search been restricted to fistula only. This was particularly relevant for communitybased prevalence studies, where the specific fistula search
only identified four studies, compared to nine when the
search was expanded to maternal and reproductive morbidity studies. Inclusion of studies that did not mention
fistula could bias the results downwards since it is not
certain that women were examined for fistula. However,

our strategy of including only studies for which a thorough


gynaecological examination was conducted should have
ensured that fistula would have been diagnosed and reported if present. Additionally, we only included studies
which elicited uterine prolapse, since this implied a
thorough examination. Because fistula is a rare condition, it is important to take account of studies that did
not find any fistulae, in the same way that studies with
negative findings are crucial in systematic reviews of
treatment effectiveness [39].
We were unable to draw solid conclusions about regional variations in the prevalence or incidence of fistula.
We only found three community based studies from
sub-Saharan Africa and four community-based studies
from South Asia (all from India or Bangladesh). Many
studies were from very select communities and it is
uncertain whether these findings are generalisable to
Africa and Asia as a whole. The heterogeneity in prevalence or incidence of fistula in community-based studies was very low however, suggesting that the prevalence
and incidence of fistula was uniformly low across all
study sites.
It is difficult to estimate the duration of fistula from
the reviewed studies because only two papers provided
an estimate. However, given that one of these from
Malawi had a median duration of three years [21], and
the other from Ethiopia [18] had a median duration of
eight years, it seems that women can live with this condition for a very long time, in some contexts. Other
studies have shown that women live with these conditions for years before presenting for fistula repair,
sometimes as long as over 20 years [10]. Additionally
there was a lack of information on the mode of delivery
and cause of the fistula in the community based studies, meaning that it is possible that some women suffered a fistula from causes other than prolonged or
obstructed labour.
The low prevalence and incidence of fistula among
women recruited in hospital is somewhat unexpected,
since women seeking care from hospitals tend to be selfselecting because they are ill. The low incidence among

Selection

Comparability Outcome

Study:

What is the case


definition? (Condition
of interest)

Representativeness of
the study population

Selection of non cases

Comparability Assessment
of cases and
of outcome
non-cases

Was study long enough


to ensure cases would
be found

Differential follow up?

Muleta et al., 2008


[18]

Obstetric fistula treated


and untreated

Population based
sample of seven
administrative
regions of rural
Ethiopia

Only women reporting


All from same
leaking examined, therefore population
it is possible that some
women may have been
counted as non cases

Sufficient physical As sample was women


exam
of reproductive age,
some women will have
only just given birth
and it is possible they
may have not yet
developed fistula

Do not state

Walraven et al.,
2001 [24]

Obstetric morbidities
including fistula

Population based
rural region

All women invited for a


physical examination

All from same


population

Sufficient physical As sample was women


exam
of reproductive age,
some women will have
only just given birth and
it is possible they may
have not yet developed
fistula

28% of sample did not


have examination

Kulkarni, 2007 [35]

Obstetric morbidities
including fistula

Population based

Included all women


examined

All from same


population

Sufficient physical Women with children at


exam
least six months
examined so assumption
is that it would be six
months postpartum

25% of sample women


did not have examination

Bhatia et al., 1997


[19]

Gynecological morbidity Population based


including fistula

Included all women


examined

All from same


population

Sufficient physical Women had exam after


exam
one year so long enough
for fistula to develop

5% lost to follow up,


6% not examined

Younis et al., 1993


[29]

Gynaecological and
related morbidities, but
do not state that they
looked for fistula

Population based

Included all women


examined

No cases

Sufficient physical Women who were ever


married and not
exam
pregnant, so it is
possible they may have
not yet developed fistula

Do not state

Deeb et al., 2003 [27] Gynaecological and


related morbidities, but
do not state that they
looked for fistula

Population based

Included all women


examined

No cases

Sufficient physical Women who were ever


9% did not have
exam
married and not pregnant, examination
so it is possible they may
have not yet developed
fistula

Al-Riyami et al., 2007


[28]

Gynaecological and
related morbidities, but
do not state that they
looked for fistula

Population based
from national
survey

Included all women


examined

No cases

Sufficient physical As sample was women of


exam
reproductive age, it is
possible they may have
not yet developed fistula

Al-Qutob, 2001 [26]

Gynaecological and
related morbidities, but
do not state that they
looked for fistula

Population based

Included all women


examined

No cases

Sufficient physical Women who were ever


10.7% did not have
exam
married and not pregnant, examination
so it is possible they may
have not yet
developed fistula

Adler et al. BMC Pregnancy and Childbirth 2013, 13:246


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Table 3 Sources of risk of bias in included studies

Do not state

Page 10 of 14

Population based, but


in Istanbul which may
not be representative
of Turkey as a whole.
Additionally only
included women who
had ever used
contraception

Included all women


examined

No cases

Sufficient physical Unclear how long women 5% did not have


exam
were followed up for
examination
after pregnancy

Tehrani et al., 2011


[34]

Gynaecological and
related morbidities,
but do not state
that they looked for
fistula

Population based

Included all women


examined

No cases

Sufficient physical All women from 18-45


119 dropped out
exam
who were not pregnant,
so it is possible some may
not have had time for
fistula to form

Ijaiya and Aboyeji,


2004 [23]

Obstetric fistula

Hospital record review


of fistula repairs with
details about reference
population

Case series of repairs

No non cases

Physical exam
and treatment

All women already had


fistula. Possible that
women missed who
did not present for
treatment

N/A

Kalilani-Phiri et al.,
2010 [21]

Obstetric fistula

Hospital record review


with details of
population it came
from, however
researchers eliminated
all cases not originating
in the region.

Case series of repairs

No non cases

Physical exam
and treatment

All women already had


fistula. Possible that
women missed who did
not present for treatment

N/A

Mabeya, 2004 [36]

Obstetric fistula

Hospital record review


of fistula repairs with
details about reference
population

Case series of repairs

No non cases

Physical exam
and treatment

All women already had


fistula. Possible that
women missed who did
not present for treatment

N/A

Vangeenderhuysen
et al., 2001 [22]

Obstetric morbidities
including fistula

Population based

Included all women


examined

All from same


population

Sufficient physical Followed up to 60 days


exam
after birth

5.7% loss to follow up

Ferdous et al., 2012


[33]

All short and long term


postpartum morbidities
including fistula

Women with morbidities


and random sample of
all women

Included all women


examined

All from same


population

Sufficient physical Examined 6-9 weeks


exam
postpartum

4.1% lost to follow up


and 6.1% did not have
examination

Fronczak et al., 2005


[20]

Obstetric morbidities
including fistula

Population based

All women examined,


but women who may
have had fistula followed
up longer

All from same


population

Sufficient physical Women feared to have


exam
fistula followed up one
month postpartum

63% did not have


examination

Filippi et al., 2007


[30]

Severe obstetric
complications
including fistula

Women with
complications overrepresented but also
had follow up of women
with uncomplicated birth

All women examined

All from same


population

Sufficient physical Women had follow up


exam
at six months

11% only had either


interview or physical
exam at six months
Page 11 of 14

Bulut et al., 1995 [25] Gynaecological and


related morbidities,
but do not state
that they looked
for fistula

Adler et al. BMC Pregnancy and Childbirth 2013, 13:246


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Table 3 Sources of risk of bias in included studies (Continued)

Filippi et al., 2010


[31]

Severe obstetric
complications
including fistula

Women with
complications overrepresented but also
had follow up of women
with uncomplicated birth

All women examined

All from same


population

Sufficient physical Women had follow up


exam
at six months

Prual et al., 1998 [32]

Severe obstetric
complications
including fistula

Women with complications All women examined


over-represented but also
had follow up of women
with uncomplicated birth

All from same


population

Sufficient physical Unclear how long women Do not state


exam
were followed up for after
pregnancy so it is possible
they may have not yet
developed fistula

32% of women did not


have follow up at six
months

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Table 3 Sources of risk of bias in included studies (Continued)

Page 12 of 14

Adler et al. BMC Pregnancy and Childbirth 2013, 13:246


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women followed in the community after admission to


hospital with near-miss obstetric morbidity (0.9 per 1000
and 1.4 per 1000) [30,31] is particularly surprising, since
this sample would have included a substantial number of
women with a prolonged and complicated labour. For
example in Benin 27.1% of women had near-miss due to
dystocia, which would have included women with both
obstructed and prolonged labour [31]. Because these
women were recruited in hospital, a timely caesarean
section may have prevented the fistula from developing.
Reduced fertility is common in women with fistula (due
to loss of vagina, amenorrhoea, not engaging in intercourse, and inability to have a live baby) [2,7,9], and it
is unlikely that women in the included incidence studies would have had a fistula before getting pregnant.
Attention should be drawn to the fact that the metaanalysis included one study from Ethiopia that had
76.4% (Figure 3) or 91.3% of the weight in communitybased studies (Figure 4), possibly inflating the pooled
prevalence estimate.
The low frequency of fistulae in any community make
survey enquiries that specifically target the counting
of fistula cases prohibitively expensive given the large
sample sizes required. Still, all community based studies
of reproductive health should explicitly ascertain and report the clinical presence or absence of fistula, even
when the sample size is small. .
In many countries in sub-Saharan Africa there is an
emphasis on building specialised fistula hospitals dedicated to the treatment of women suffering from fistula.
Given the rarity of the condition and the high level of
skills and training required for fistulae surgery, the results
of this review suggest that the majority of the resources
will always be better placed on prevention rather than
cure. Strengthening maternal health services, creating
conditions for better transportation and communication
networks and training of local providers into the management of emergency complications, including with caesarean sections, would have the additional effect of providing
care for other causes of maternal and perinatal mortality
and morbidity.
The relative rarity of fistula should not detract from
their public health importance. The estimated 6000 new
cases of fistula per year in sub-Saharan Africa and South
Asia are a painful testament to the continued failure of
health systems to manage labour complications effectively. Caesarean sections remain inaccessible to a large
number of women in sub-Saharan Africa [40]. Delays in
accessing caesarean sections, faulty techniques and lack
of caesarean sections all contribute to the burden of fistula. The fact that fistula have virtually disappeared in high
income countries suggest that they are entirely preventable. Given the seriousness of the condition, and the devastating consequences of fistula for women and their

Page 13 of 14

families, efforts should also be made to find these women


and treat them.

Conclusions
Our study is the most comprehensive study of the burden of fistula to date, including study sources not generally used. Our findings suggest that the prevalence and
incidence of fistula is relatively low. The low burden of
fistula should not detract from their public health importance, however, given the preventability of the condition, and the devastating consequences of fistula. Future
studies of fistula should include a description of the
study population with defined denominators.
Competing interests
The authors declare that they have no competing interests.
Authors contributions
AJA conducted search, extracted data, did analysis and wrote first draft of
paper CR advised on methodology and commented on drafts CC
commented on drafts and helped in writing of paper VF helped design
study and commented on paper. All authors read and approved the final
manuscript.
Acknowledgements
The authors would like to thank Doris Chou, Lale Say, and Herbert Peterson
for their comments.
Funding
This research was funded through a grant made to the Child Health
Epidemiology Reference Group (CHERG) by the Bill and Melinda Gates
Foundation.
Received: 13 June 2013 Accepted: 9 December 2013
Published: 30 December 2013
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doi:10.1186/1471-2393-13-246
Cite this article as: Adler et al.: Estimating the prevalence of obstetric
fistula: a systematic review and meta-analysis. BMC Pregnancy and
Childbirth 2013 13:246.

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